Literature DB >> 16848116

Cholesteryl ester storage disease in a young child presenting as isolated hepatomegaly treated with simvastatin.

Buket Dalgiç1, Sinan Sari, Mehmet Gündüz, Fatih Ezgü, Leyla Tümer, Alev Hasanoğlu, Gülen Akyol.   

Abstract

Cholesteryl ester storage disease (CESD) is an autosomal recessive disorder resulting from lysosomal acid lipase deficiency and is usually characterized by hepatomegaly and hyperlipidemia. This paper reports a two-year-old boy who had hepatosplenomegaly, hyperlipidemia and hypertransaminasemia determined incidentally. The liver biopsy sample was orange-yellow in appearance. Microscopically, microvesicular steatosis and birefringent crystals were seen in liver biopsy. The diagnosis of CESD was confirmed by the reduced human acid lipase activity in peripheral leukocytes. Simvastatin therapy was given and tolerated without side effects. Our patient is the youngest reported case in the literature treated with 3-hydroxy 3-methyl glutaryl (HMG) CoA reductase inhibitor.

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Year:  2006        PMID: 16848116

Source DB:  PubMed          Journal:  Turk J Pediatr        ISSN: 0041-4301            Impact factor:   0.552


  5 in total

1.  A case of abdominal pain with dyslipidemia: difficulties diagnosing cholesterol ester storage disease.

Authors:  S J Cameron; U Daimee; R C Block
Journal:  Eur Rev Med Pharmacol Sci       Date:  2015       Impact factor: 3.507

2.  Novel mutation in a patient with cholesterol ester storage disease.

Authors:  Patrick Lin; Sheela Raikar; Jennifer Jimenez; Katrina Conard; Katryn N Furuya
Journal:  Case Rep Genet       Date:  2015-02-05

Review 3.  Targeting Wolman Disease and Cholesteryl Ester Storage Disease: Disease Pathogenesis and Therapeutic Development.

Authors:  Francis Aguisanda; Natasha Thorne; Wei Zheng
Journal:  Curr Chem Genom Transl Med       Date:  2017-01-30

4.  Effect of a common missense variant in LIPA gene on fatty liver disease and lipid phenotype: New perspectives from a single-center observational study.

Authors:  Andrea Pasta; Paolo Borro; Anna Laura Cremonini; Elena Formisano; Giulia Tozzi; Stefano Cecchi; Raffaele Fresa; Sara Labanca; Afscin Djahandideh; Samir Giuseppe Sukkar; Antonino Picciotto; Livia Pisciotta
Journal:  Pharmacol Res Perspect       Date:  2021-10

Review 5.  Lysosomal acid lipase deficiency in pediatric patients: a scoping review.

Authors:  Camila da Rosa Witeck; Anne Calbusch Schmitz; Júlia Meller Dias de Oliveira; André Luís Porporatti; Graziela De Luca Canto; Maria Marlene de Souza Pires
Journal:  J Pediatr (Rio J)       Date:  2021-05-06       Impact factor: 2.990

  5 in total

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